Prader-Willi syndrome (PWS) is a complex neurodevelopmental disorder resulting from loss of actively transcribed DNA from the PWS region of the paternally derived chromosome 15. Insights from the study of individuals with PWS have impacted our understanding of genetic imprinting, the role of noncoding RNAs in human disease, mechanisms of obesity, genetics of neuropsychiatric illness, and normal neurodevelopment. However, the underlying basis of the PWS phenotype remains poorly understood, despite the fact that the PWS critical region was defined more than twenty-five years ago. In addition, while better diagnostic tests and early intervention have led to improved outcomes, it has become clear that additional challenges such as mental illness, sleep disturbances, and endocrine dysfunction are significant but understudied problems. The organizers are seeking partial funding for two biennial workshops to develop a coordinated research strategy for PWS, which will include the identification of high priority research goals and the development of actionable steps towards achieving those goals. The first meeting will be held on the NIH campus in early November 2009, and will include approximately 50 scientists with expertise in the broad range of disciplines relevant to PWS. This workshop is unique in that it will bring together basic and clinical scientists from diverse backgrounds to address unresolved problems in PWS. Workshop goals include defining the challenges and opportunities in PWS research through the delineation of high priority, unresolved basic science and clinical questions;determining the resources and research tools needed to address these questions;identifying of new areas of cross-disciplinary research with a plan to promote strategic collaborations;and effectively disseminating the recommendations that arise from the workshop. Future meetings will assess progress on the goals established in the initial workshop, and revise and advance the PWS Research Strategy. PUBLIC HEALTH RELEVANCE: The Prader-Willi Syndrome Research Strategy Workshop will bring together leading scientists from the diverse fields of significance to Prader-Willi syndrome (PWS), with the goal of developing a research strategy to advance the science of PWS. Participants will prioritize research questions and identify opportunities and needs. Recommendations will be made with respect to resource development, collaborative opportunities, and training mechanisms to facilitate attainment of research goals. Advancement of research for this rare disorder will shed light on common public health concerns including eating disorders, obesity, sleep disorders, and mental health.